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LETTER TO EDITOR
Year : 2014  |  Volume : 62  |  Issue : 3  |  Page : 308-310

Cerebral venous sinus thrombosis presentation in severe scrub typhus infection: A rare entity


1 Department of Neurological Sciences, Christian Medical College, Vellore, TamilNadu, India
2 Department of Neurological Sciences; Section of Neurosurgery, Christian Medical College, Vellore, TamilNadu, India
3 Department of Clinical Microbiology, Christian Medical College, Vellore, TamilNadu, India

Date of Submission11-Jun-2014
Date of Decision30-Jun-2014
Date of Acceptance15-Jun-2014
Date of Web Publication18-Jul-2014

Correspondence Address:
Subhransu S Jena
Department of Neurological Sciences, Christian Medical College, Vellore, TamilNadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.136991

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How to cite this article:
Jena SS, Mathew A, Sanjith A, Ajith S, Nair BR, Prakash J. Cerebral venous sinus thrombosis presentation in severe scrub typhus infection: A rare entity. Neurol India 2014;62:308-10

How to cite this URL:
Jena SS, Mathew A, Sanjith A, Ajith S, Nair BR, Prakash J. Cerebral venous sinus thrombosis presentation in severe scrub typhus infection: A rare entity. Neurol India [serial online] 2014 [cited 2019 Aug 23];62:308-10. Available from: http://www.neurologyindia.com/text.asp?2014/62/3/308/136991


Sir,

Scrub typhus is a common re-emerging infection in the Indian subcontinent. [1] The severity of infection can range from mild symptoms and signs to multi organ failure. Severe scrub infection can have varied presentation: Pulmonary edema, interstitial pneumonia, congestive heart failure, circulatory collapse, glomerulonephritis, and central nervous system (CNS) dysfunction, including confusion, delirium, and seizures. This report describes a case of scrub typhus with cerebral sinus thrombosis (CST), an unreported complication of scrub typhus.

A 48-year-old man presented with fever headache and vomiting of five days duration followed by right side focal seizures and altered sensorium for 2 days. One week before admission, he had symptoms of an upper respiratory tract infection with productive cough and sore throat. At admission, neurological examination revealed Glasgow Coma Scale (GCS) score of 11/15 (E3V3M5), bilateral papilloedema, paucity of right side movement with hypoesthesia, and an extensor plantar response on right side. He had a high grade persistent fever (~39°C) and no rash or eschar. Laboratory findings revealed thrombocytopenia (55,000/mm 3 ); hematuria, elevated creatinine (2.73 mg/dl); urea (62 mg/dl); elevated aspartate aminotransferase (183 IU/L) and alanine aminotransferase (79 IU/L); and serum Creatine phosphokinase and Lactate dehydrogenase were 20,842 U/L and 1906 U/L respectively. Fever work-up profile was non-contributory. Work-up for disseminated intravascular coagulation (DIC) was within normal limits: Prothrombin time (11.9 seconds), activated partial thromboplastin time (25.7 seconds), and fibrinogen level (372.7 mg/dl). IgM and IgG serology for dengue was negative. Diagnosis of scrub typhus was confirmed by serum IgM ELISA. Chest X-ray showed bilateral pulmonary infiltrates suggestive of interstitial pneumonitis. Magnetic resonance imaging (MRI) of brain showed thrombosis of the anterior portion of superior saggitial sinus with hemorrhagic venous infarct in left frontal lobe and a midline shift of 4 mm to right side [Figure 1]. A comprehensive thrombotic work-up was normal. Ham's test and sucrose lysis test were negative. Genetic markers for thrombosis like factor V Leiden mutation, methylene tetra hydro folate reductase, and prothrombin gene polymorphism were negative.
Figure 1: (Day-1): Magnetic resonance imaging/magnetic resonance venography (MRI/MRV) of the brain - T2, T1W images reveals acute hemorrhagic venous infarct and swelling in the left high frontal region; midline shift to the right with mild subfalcine herniation, MRV demonstrates thrombosis in anterior part of the superior sagittal sinus extending into the cortical vein on the left

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He was treated with doxycycline, azithromycin, unfractionated heparin and anti-edema measures. On day-3 of admission, his sensorium worsened to a GCS score of 7/15 with radiologically detoriation as well, and emergency left front otemporoparietal decompressive hemicraniectomy was done. Intra-operatively, the brain was tense, and multiple thrombosed cortical veins were seen. Post-operatively, he was weaned off ventilator. He developed acute kidney injury and hypotension which were managed successfully. After comprehensive rehabilitative and medical therapy at 6-months follow-up, he had improved to a modified Rankin scale of 2. Repeat brain MRI showed marked improvement with complete recanalization of the superior sagittal sinus and reduction in size of the left high frontal hematoma [Figure 2].
Figure 2: (6 months later): Reducing extension and degree of enhancement of previous subacute venous infarction involving left frontal lobe, suggestive of improvement. Complete recanalization of the superior sagittal sinus

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The spectrum of neurological complications in scrub typhus include: Aseptic meningitis, meningoencephalitis, cerebellitis [2] , myelitis [3] , cerebral hemorrhage [4] , and cerebral infarction. [5] Autopsy study has revealed punctate focal brain hemorrhages. [3]

The cytokines released by acutely inflamed vascular endothelial cells secondary to invasion by Orientia tsutsugamushi damage endothelial integrity causing fluid leakage. There is localized platelet aggregation, polymorphs, and monocyte proliferation, leading to focal occlusive and angiitis causing micro infarcts in various tissues. This localized process can also cause venous thrombosis and peripheral gangrene. [6] The brain pathology reveals a diffuse or focal mononuclear cell exudate in the leptomeninges and typhus nodules (clusters of microglial cells) in the parenchyma.

The characteristic eschar seen in scrub typhus infection, indicative of the vector bite, is present in less than 50% of patients reported from India. [7] Papilledema as seen in the present patient was reported in the earlier studies. [8] Thrombocytopenia and DIC may aggravate the extent of the cerebral hemorrhage. Abnormal chest radiographs have been reported in about 70% of patients. Hepatocellular dysfunction in scrub typhus is transient and usually normalises within 2 weeks. [9] We used an Enzyme-linked immunosorbent assay (Scrub Typhus Group ELISA Kit InBios, Seattle, WA, USA) test to detect IgM antibodies to Orientia tsutsugamushi. This kit uses a 56-kDa recombinant cocktail of antigens (Karp, Gilliam, Kato and TA763) and has diagnostic specificity and sensitivity of ~90% and provides positive results within 3-4 days after the onset of illness. Ig M ELISA test has 86.5% sensitivity and 97.5% specificity. [10]

Various systemic or CNS infections are possibly risk factors for CST. In this patient, risk factors for thrombosis, such as drugs or hereditary hypercoagulable state or local and systemic infections other than scrub typhus were not present. We postulate that the disseminated endothelial damage causing cerebral vasculopathy and perivasculitis of the capillaries and venous stasis due to increased intracranial pressure as the most likely mechanisms of sinus thrombosis.

 
  References Top

1.Kelly DJ, Fuerst PA, Ching WM, Richards AL. Scrub typhus: The geographic distribution of phenotypic and genotypic variants of Orientia tsutsugamushi. Clin Infect Dis 2009;48 Suppl 3:S203-30.  Back to cited text no. 1
    
2.Silpapojakul K, Ukkachoke C, Krisanapan S, Silpapojakul K. Rickettsial meningitis and encephalitis. Arch Intern Med 1991;151:1753-7.  Back to cited text no. 2
    
3.Kim DE, Lee SH, Park KI, Chang KH, Roh JK. Scrub typhus encephalomyelitis with prominent focal neurologic signs. Arch Neurol 2000;57:1770-2.  Back to cited text no. 3
    
4.Shih-Hsien Y, Lih-Shinn W, Chung-Chao L, Yu-Huai H. Scrub typhus complicated by intracranial hemorrhage- A case report. Tzu Chi Med J 2005;17:111-4.  Back to cited text no. 4
    
5.Kim JH, Lee SA, Ahn TB, Yoon SS, Park KC, Chang DI, et al. Polyneuropathy and cerebral infarction complicating scrub typhus. J Clin Neurol 2008;4:36-9.  Back to cited text no. 5
    
6.Cowan G. Rickettsial diseases: The typhus group of fevers: A review. Postgrad Med J 2000;76:269-72.  Back to cited text no. 6
    
7.Chrispal A, Boorugu H, Gopinath KG, Prakash JA, Chandy S, Abraham OC, et al. Scrub typhus: an unrecognized threat in South India - clinical profile and predictors of mortality. Trop Doct 2010;40:129-33.  Back to cited text no. 7
    
8.Varghese GM, Mathew A, Kumar S, Abraham OC, Trowbridge P, Mathai E. Differential diagnosis of scrub typhus meningitis from bacterial meningitis using clinical and laboratory features. Neurol India 2013;61:17-20.  Back to cited text no. 8
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9.Yang CH, Hsu GJ,Peng MY, Young TG. Hepatic dysfunction in scrub typhus. J Formos Med Assoc 1995;94:101-5.  Back to cited text no. 9
    
10.Prakash JA, Abraham OC, Mathai E. Evaluation of tests for serological diagnosis of scrub typhus. Trop Doct 2006;36:212-3.  Back to cited text no. 10
    


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